PEDIATRIC/CRANIOFACIAL Outcomes Article
Comparative Effectiveness Studies Examining Patient-Reported Outcomes among Children with Cleft Lip and/or Palate: A Systematic Review Kavitha Ranganathan, M.D. Christian J. Vercler, M.D. Seth A. Warschausky, Ph.D. Mark P. MacEachern Steven R. Buchman, M.D. Jennifer F. Waljee, M.D., M.S. Ann Arbor, Mich.
Background: Health care policy makers are increasingly encouraging comparative effectiveness research. Little is known regarding comparative studies among children with cleft lip and/or palate. Cleft lip and/or palate profoundly influences self-perception and social functioning, and patient-reported outcomes provide a unique perspective on the success of reconstruction. The purpose of this study was to systematically review the literature regarding patient-reported outcomes among patients with cleft lip and/or palate. Methods: The authors reviewed articles from MEDLINE, Embase, and PsycInfo that examined the use of patient-reported outcome instruments for cleft lip and/or palate. Studies of patients with cleft lip and/or palate across any age that described the use of patient-completed measures in patient and control populations were included. A research librarian confirmed the search, and two independent, blinded reviewers performed full-text review. Results: The authors identified 1979 articles and selected 30 for inclusion. Forty-two different assessment tools were used to analyze factors such as selfesteem, behavior, and social support. The Strengths and Difficulties Questionnaire was most commonly used (n = 7), followed by the Childhood Experience Questionnaire (n = 5), and the Satisfaction with Appearance survey (n = 4). Barriers to analysis included lack of standardization of survey administration, effect of publication bias, and variations in patient populations between individual studies. Conclusions: Comparative studies of patient-reported outcomes among patients with cleft lip and/or palate are infrequent. Many instruments exist to measure patient-reported outcomes in this population, but no specific standard exists. Identifying efficient and targeted forms of instrument selection and administration will enhance comparative studies among children with cleft lip and/or palate. (Plast. Reconstr. Surg. 135: 198, 2015.) CLINICAL QUESTION/LEVEL OF EVIDENCE: Diagnostic, III.
left lip and/or palate is among the most common craniofacial anomalies, affecting over 5000 children born each year in the United States.1 The manifestations associated with cleft lip and/or palate begin at birth and extend into adulthood, ranging from imperceptible scars to significant facial deformity. Although there are innumerable reconstructive techniques, clear From the Section of Plastic Surgery, Department of Surgery, the Department of Physical Medicine and Rehabilitation, and the Taubman Health Sciences Library, University of Michigan Health System. Received for publication March 12, 2014; accepted June 6, 2014. Copyright © 2014 by the American Society of Plastic Surgeons DOI: 10.1097/PRS.0000000000000825
evidence to support a single approach is lacking.2 Given accelerating health care costs in the United States, it is essential to identify those treatment strategies that yield optimal outcomes.3 Disclosure: The authors have no financial interest to declare in relation to the content of this article. Supplemental digital content is available for this article. A direct URL citation appears in the text; simply type the URL address into any Web browser to access this content. A clickable link to the material is provided in the HTML text of this article on the Journal’s Web site (www. PRSJournal.com).
Volume 135, Number 1 • Cleft Lip and/or Palate Comparative Study Comparative effectiveness research has evolved to illuminate those aspects of health care that are most effective, and describes research that explicitly includes comparison studies to evaluate the value of existing treatments.4 Although there is substantial enthusiasm and financial support for comparative effectiveness research from both public and private sectors, comparative studies are sparse, particularly in surgical disciplines.5–7 Understanding outcomes following reconstruction through comparative studies allows us to critically appraise current treatment options, longitudinally assess patient recovery over time, and identify benchmarks by which quality of care can be improved. Patient-reported outcomes following reconstruction for cleft lip and/or palate describe aspects of health obtained directly from patients and can augment outcomes assessments based on clinical events in several ways.8–11 First, information obtained directly from patients provides a more holistic and specific perspective of recovery.12 For example, many patients report overall satisfaction with the operative results despite the occurrence of complications.13–15 Furthermore, patient-reported outcomes capture the benefits of surgery beyond physiologic markers, and are often the outcomes most important to patients.16–19 Finally, measuring effectiveness using patientreported outcomes aligns with broader goals of recent health care reform and holds providers accountable for patient experiences.20 The difficulty with using patient-reported outcomes in the cleft lip and/or palate population is important to acknowledge. Congenital anomalies require long-term monitoring, which is inherently fraught with challenge. Although proxy measures can be used during infancy, the accuracy of these measures is questionable. Thus, one of the only ways to address these challenges is to use serial assessment strategies to monitor progress over time. Although ideally patient and proxy measures can be used concomitantly, the lack of a current standard leaves this option challenging in a practical setting. Thus, we believe that it is best to first identify the proper tools and administration strategies in a setting that minimizes confounding variables, and then focus on designing serial evaluation strategies. The purpose of this study was to systematically review the literature to identify comparative effectiveness studies of patient-reported outcomes following cleft lip and/or palate reconstruction to examine aesthetic, functional, social, and psychological outcomes. We hypothesize that although many instruments are available to assess these
specific outcomes, the limited use of control groups, lack of uniform data analysis, and difficulty in isolating confounding factors will limit the overall generalizability and utility of currently available measures.
MATERIALS AND METHODS Literature Search We performed a comprehensive search of the PubMed, Embase, and PsycINFO databases from inception of each database to August of 2013 according to Preferred Reporting Items for Systematic Reviews and Meta-Analyses guidelines. A medical librarian formulated the search strategy after consultation with the lead authors (J.F.W. and K.R.). Both English and non-English articles were eligible for inclusion. A set of three key articles, which were identified by the authors before the search, were used as a primary source of relevant text words and controlled terms.1,21,22 The key articles were pooled together and run against the result sets to ensure the searches were capturing appropriate citations. In PubMed and Embase, concepts were approached using a combination of controlled vocabulary terms (Medical Subject Headings and EMTREE) and title/ abstract text words. In both databases, a filter designed to exclude animal studies was applied. The PsycINFO search was similarly constructed, but did not include an animal study filter. No other filters or limits were added to the searches. The authors reviewed reference lists of key articles to supplement the database searches and identify further studies. Two separate screeners evaluated the titles, abstracts, and/or full text of all eligible articles retrieved in this search. In total, the database searches retrieved 1970 citations. These citations were imported into Endnote X5, which identified and eliminated 601 duplicates. The 1369 that remained were moved to an Excel spreadsheet (Microsoft Corp., Redmond, Wash.) before the formal review. The complete search strategies are available in Figure 1. Inclusion and Exclusion Criteria We included all studies that met the following criteria: (1) conducted among cleft lip and/ or palate patients with or without associated conditions, (2) identified a normative or specific comparison group, and (3) directly evaluated patient-reported outcomes rather than only proxy or parent-reported outcomes. Articles
Plastic and Reconstructive Surgery • January 2015
Fig. 1. Flow chart of studying methodology based on inclusion and exclusion criteria. PRO, patient reported outcome; CLCP, cleft lip and/or palate.
were excluded if they did not perform a specific analysis of cleft lip and/or palate patients, used parent/caregiver proxy measures instead of patient-reported data, did not include a specific cleft or normative comparison group, or were not available in full-text format. Articles were reviewed for quality using the U.S. Preventive
Services Task Force guidelines. Patients of all age groups were included given that age is an important variable to consider in survey administration. Data Collection and Quality Assessment Patient demographic information, cleft type, survey type, method of administration,
Volume 135, Number 1 • Cleft Lip and/or Palate Comparative Study characteristics of the control group, and documented difficulties related to survey use were recorded for each study that fit the inclusion criteria. We also extracted the results of each survey and the exact construct measured with regard to specific outcomes of aesthetics, social functioning, mood disorders, and health-related quality of life. Each article was assessed for quality using the U.S. Preventive Services Task Force guidelines.23 These guidelines examine specific factors of each article such as differential loss to follow-up, quality of outcome measures, definition of patients and interventions, and adjustment for confounding variables. Using this information, each study is given a rating of good, fair, or poor. The authors reviewed each study according to these criteria.
RESULTS Summary of Literature Review Using our search criteria, a total of 1970 studies were identified. An additional nine citations were added after examining systematic reviews performed on cleft lip and/or palate patients. After title, abstract, and full-text analysis, 30 studies met final inclusion criteria. Table 1 lists information regarding study characteristics. Of the included studies, sample sizes ranged from 18 to 754 subjects consisting of patients from 12 different countries. Age groups ranged from 2 to 65 years. Approximately 42 different assessment tools were used to analyze factors such as selfesteem, behavior, coping mechanisms, support, intelligence, and speech. The number of items in the measures ranged from five to 567. The patient population for 18 studies consisted of cleft lip, cleft palate, and cleft lip and cleft palate patients.24–41 Cleft palate and cleft lip and cleft palate patients only were grouped together in three studies.42–44 Cleft lip and cleft palate patients were included in seven studies.45–51 Only one study consisted of cleft lip patients alone.52 Cleft subtypes were not clearly stated in the remainder of the studies.53 Normative control groups were used by seven studies.24,25,33,36,38,45,47 In contrast, 15 studies used specific control groups.26–31,37,40,46,48–53 A total of seven studies compared various subgroups within the sample population.32,34,35,41–44 (Tables 3 and 4) (see Appendix, Supplemental Digital Content 1, for content analysis, http://links.lww.com/PRS/ B188). Comparative studies used generic survey measures rather than cleft-specific instruments. In contrast, studies that used cleft-specific measures
(e.g., Cleft Evaluation Profile) did not use comparative analyses. The Strengths and Difficulties Questionnaire was most commonly used (n = 7), followed by the Childhood Experience Questionnaire (n = 5) and the Satisfaction With Appearance survey (n = 4) (Table 2). The Oral Health Impact Profile was the most specific instrument used and has been validated specifically in the craniofacial population.1,36 Aesthetic Outcomes Aesthetic considerations were most commonly analyzed using the Satisfaction With Appearance questionnaire.24,25,32,43 The Strengths and Difficulties Questionnaire, Self-Perception Profile for Adolescents, and Multidimensional Self-Esteem Inventory were also used to evaluate body image and self-perception related to appearance in this review.25,33,50,51 Some studies have demonstrated that female sex, adolescence, and visibility of the cleft lead to lower social perceptions, self-esteem, and satisfaction with appearance.32,35,41 Others, however, have found that patients with a cleft lip and/or palate report significantly greater satisfaction with various aspects of their facial appearance compared with adolescents with no facial disfigurement.25,30,33,45,50 Social Outcomes Social functioning as defined by cognitive perceptions of relationships, emotional functioning, and adjustment levels were most commonly measured using the Childhood Experience Questionnaire (n = 5 studies), as detailed in Table 2.24,25,32,43 Similar to outcomes on aesthetic measures, significant variability exists in the results of studies examining communication and social functioning among patients with clefts compared with the general population. Although some studies show that patients with clefts have greater difficulty with communication and social functioning, others demonstrate that adolescents with a cleft have more positive social and self-perceptions compared with control patients.26–28,36,37,46–48,52,53 Still others show no difference in socioeconomic variables, quality of life, and psychosocial adjustment between patients with and without clefts.40,48,49,51,53 Reasons to explain such variability in outcomes include difficulty in detecting cleft-specific differences using general surveys, overestimation of the quality of social interactions, by patients with clefts, and increased level of support offered to patients with cleft in the form of frequent follow-up for management of the cleft. This
Plastic and Reconstructive Surgery • January 2015 Table 1. Summary of Patient-Reported Outcome Measures Administered to Cleft Lip and/or Palate Patients Name of Survey
No. of Times Used
Strengths and Difficulties Questionnaire
Childhood Experience Questionnaire Satisfaction With Appearance
Social Avoidance and Distress Scale Culture Free Self-Esteem Inventories Self-Perception Profile for Adolescents 36-Item Short-Form Health Survey Beck Depression Inventory KIDCOPE Satisfaction With Life Scale Primary Self-Concept Inventory State-Trait Anxiety Inventory for Children
3 3 3 2 2 2 2 2 2
Taylor Manifest Anxiety Scale Minnesota Multiphasic Personality Inventory
Piers-Harris Children’s Self-Concept Scale Participation in Everyday Life Communication Questionnaire Chinese Miller Behavioral Style Scale Child Depression Inventory Schedule for Affective Disorders and Schizophrenia for School-Age Children-Present and Lifetime Version Ankara Articulation Test Wechsler Intelligence Scale for Children-Revised
Psychosocial adjustment, behavior, peer relations Social experiences, psychosocial adjustment Social and behavioral components of body image in relation to self-perceptions Quantification of social anxiety Self-esteem Self-esteem and self-worth Functional health and well-being Quantification of levels of depression Measurement of coping strategies Subjective well-being, coping strategies Self-concept Definition/quantification of state versus trait anxiety Nature and degree of anxiety in children Identification of personal, social, behavioral concerns Generalized self-concept Communicative competency
1 1 1
Coping mechanisms Presence and severity of depressive symptoms Social functioning, behavioral characteristics
Phoneme acquisition skills Intelligence
Beck Anxiety Inventory Personality Inventory for Children
Hopkins Symptom Checklist-25 Oral Health Impact Profile-14
Rosenberg Self-Esteem Scale/Self-Esteem Index Youth Self-Report/Young Adult Self-Report KINDL Quality of Life Questionnaire
1 1 1 1
Severity of anxiety Emotional, behavioral, cognitive, and interpersonal adjustment Symptoms of anxiety and depression Levels of dysfunction, discomfort and disability associated with oral disorders Global self-esteem Emotional/behavioral problems Perceived quality of life Satisfaction and well-being
Pediatric Behavior Scale
Reynolds Child Depression Scale/Adolescent Depression Scale Pictorial Scale of Perceived Competence and Social Acceptance for Young Children Rand-36 Multidimensional Self-Esteem Inventory Coping Inventory for Stressful Situations WHOQOL-BREF Questionnaire General Health Questionnaire-28
Internalized and externalized aspects of behavior Depression
1 1 1 1 1
Eysenck Personality Inventory Carter Self-Perception Profile for Children/ Adolescents Social Support Scale for Children and Adolescents
Quality of life Self-esteem Coping mechanisms and adjustment Quality of life Quantification of risk of developing a psychiatric disorder Personality traits Self-perception
Child Health and Illness Profile
variability has been demonstrated using various questionnaires, including the Strengths and Difficulties Questionnaire, KIDCOPE, Childhood
How children felt support from environmental factors Overall health and well-being
No. of Items 25 20 14 28 60 28 36 21 9 5 18 40 37 567 60 59 8 27 82 — 6 verbal subscales and 5 performance subscales 21 275 25 14 10 103/132 24 One global, six life domain question sets 30 30 24 36 116 48 26 60 (28-item version) 57 36 8 107
Experience Questionnaire, 36-Item Short Form Health Survey (“SF-36”), and Satisfaction With Appearance assessments.24,25,45,47
Strengths and Difficulties Questionnaire-2
4170 participants, age-matched
5494 students in Australia
1500 German schoolchildren aged 8–12 yr 1384 age- and sexmatched data retrieved from the national SF-36 database
Foo et al.36
Gussy and Kilpatrick.45
Kramer et al.38
Mani et al.47
Hopkins Symptom Checklist, Self-Perception Profile for Adolescents
1832 normal adolescents
Feragan et al.33
Schoolchildren with orofacial clefts had similar or higher scores than observed in an age-matched unaffected control group of German children in all dimensions. In specific subgroups, children with CLCP had lower scores in dimensions measuring family and everyday activity than the children with CL or CP. Quality of life for unilateral CLCP patients was similar to those of the normative population in six of seven SF-36 subscales. Mental health subscale was significantly lower for cleft patients compared with normative data.
The study group did not differ from the general adolescent population in global or total self-concept. In terms of specific self-concept scales, the study group had more positive perceptions in the areas of physical ability, physical appearance, opposite-sex relations, and parent relations.
Adolescents with a visible cleft reported more positive perceptions of close friendships, higher levels of social acceptance, more positive self-perceptions of appearance, and less emotional distress. Social acceptance was significantly associated with physical appearance and with depressive symptoms. Physical appearance and depressive symptoms also showed significant associations. Close friendships were related to physical appearance (significant) and to depressive symptoms (not significant). The cleft patients scored lower on vitality and mental health functions with the SF-36 compared with the South Australian norms. OHIP-14 findings indicated markedly worse (nearly three times worse than population norms) oral health impact among cleft patients. No significant sex differences.
Adolescents with a cleft reported equivalent levels of adjustment to the general population. The most popular strategies reported by adolescents were resignation, social support, and distraction. Younger adolescents reported using social support significantly more frequently than did older adolescents. The current sample reported significantly greater satisfaction with various aspects of their facial appearance compared with adolescents with no facial disfigurement. The exception to this was the lip: the cleft group reported significantly less satisfaction with their lips. Social experiences determine adjustment; more negative experiences means = worse adjustment strategies. Male sex and use of social withdrawal as coping strategy were predictive of poor adjustment. Cleft type alone was not predictive of adjustment.
SDQ, Strength and Difficulties Questionnaire; CEQ, Childhood Experience Questionnaire; SF-36, 36-Item Short-Form Health Survey; OHIP-14, Oral Health Impact Profile-14; CLCP, cleft lip and/or palate; CL, cleft lip; CP, cleft palate.
Quality of life varies with gender and age among adults treated for unilateral cleft lip and palate
General health-related quality of life and oral health impact among Australians with cleft compared with population norms; age and gender differences The self-concept of adolescents with cleft lip and palate: A pilot study using a multidimensional/hierarchical measurement instrument Quality of life in school-age children with orofacial clefts and their families
Strengths and Difficulties, KIDCOPE, Childhood Experience Questionnaire
SDQ, 5000 British patients; KIDCOPE, 731 British adolescents; CEQ, 743 adolescents)
Berger and Dalton24
Coping with a cleft II: Factors associated with psychosocial adjustment of adolescents with a cleft lip and palate and their parents Adolescents with and without a facial difference: The role of friendships and social acceptance in perceptions of appearance and emotional resilience
Survey Strengths and Difficulties, KIDCOPE, Childhood Experience Questionnaire, Satisfaction with Appearance
Normative Group Characteristics SDQ, 5000 British patients; KIDCOPE, 731 British adolescents; CEQ, 743 adolescents
Berger and Dalton25
Coping with a cleft: psychosocial adjustment of adolescents with a cleft lip and palate and their parents
Table 2. Summary of Studies Using Normative Control Populations as Comparison Groups to Cleft Lip and/or Palate Patients
Volume 135, Number 1 • Cleft Lip and/or Palate Comparative Study
Child Depression Inventory, State-Trait Anxiety Inventory for Children, Piers-Harris Children’s Self-Concept Scale: Schedule for Affective Disorders and Schizophrenia for School-Age Children-Present and Lifetime Version, Ankara Articulation Test, Wechsler Intelligence Scale for ChildrenRevised, Beck Anxiety Inventory, Beck Depression Inventory
Normal patients Social Avoidance and Distress scheduled to Scale, Satisfaction with Life undergo third molar Scale, Culture Free Self-Esteem extraction; normaInventory, Chinese Miller tive values based Behavioral Style Scale on previously cited studies
Social Avoidance and Distress Scale, Satisfaction With Life Scale, Culture Free Self-Esteem Inventory
Nine normal patients undergoing orthognathic surgery for maxillofacial deformities
CLCP adult patients of either sex tended to have greater satisfaction with life, higher social avoidance, and lower parental and general self-esteem than the adolescent group. Chinese adults with clefts did not differ between normal adults in terms of satisfaction with life. CLCP adolescent patients exhibited significantly lower general and social self-esteem than the control group. The CLCP adolescent patients exhibited higher parental self-esteem than the Chinese non-CLCP subjects. In CLCP patients, children with SAD had significantly lower PHCSCS-physical appearance subscale and PHCSCS-mental state subscale scores than the children without SAD. Children with SAD had significantly fewer surgical operations than the children without SAD. The children with psychopathologic conditions had significantly more severe dentofacial deformity. Teasing over appearance was significantly associated with depressive disorders.
Patients with cleft lip, cleft palate, or CLCP had low social, personal, and total self-concept compared with controls. No differences were found in terms of intellectual domain between control and cleft groups. Between cleft groups, CLCP scored lowest on total, intellectual, and social domains, and no differences were noted between cleft lip and cleft palate patients. CLCP patients were generally happier, and had a higher level of parental support than normal patients suffering from dentofacial deformities at baseline. Cleft patients undergoing distraction had a higher level of social avoidance tendency and faced more distress than those in the conventional group.
Primary Self-Concept Inventory
First graders in North Carolina (age- and sex-matched)
CLCP boys had poorer peer relations according to the selfreported SDQ-1 score (not statistically significant).
SDQ, Participation in Everyday There were no significant differences between cleft and control Life Communication Questiongroup on the SDQ. On the PIELCQ, however, CLCP kids had naire greater difficulty in the social environment. In general for both groups, increased problems with peers correlated negatively with daily activity, and hyperactivity correlated positively with increased mood and sleep quality.
34 age- and sexmatched controls
43 healthy, normal, age- and sexmatched boys
Psychiatric assessment of Demir et al.31 40 patients age- and sex-matched to cleft children with nonsyngroup dromic cleft lip and palate
The early psychological Cheung adjustment of cleft et al.29 patients after maxillary distraction osteogenesis and conventional orthognathic surgery: A preliminary study Psychological profile of Cheung Chinese with cleft lip et al.30 and palate deformities
Social function in boys Boes et al. with cleft lip and palate: Relationship to ventral frontal cortex morphology Psychosocial functioning Brand et al.27 and sleep patterns in children and adolescents with cleft lip and palate (CLP) compared with healthy controls Self-concept of early Broder and primary school age Strauss28 children with visible or invisible defects
Specific Control Population Characteristics
Table 3. Summary of Studies Using Specific Samples of Noncleft Patients as Comparison Groups to Cleft Lip and/or Palate Patients
Plastic and Reconstructive Surgery • January 2015
Prevalence of psychiatric Yunusa and morbidity using GHQObembe52 28 among cleft lip patients in Sokoto Social acceptance and Slifer et al.40 facial behavior in children with oral clefts
43 age-, socioeconomic-, sex-matched people recruited from the population 48 participants matched Multidimensional Self-Esteem Individuals with CLCP had higher self-esteem with respect to for age, location, Inventory, Coping Inventory body functioning and scored higher in defensive selfsocioeconomic status, for Stressful Situations, WHOenhancement compared with controls. There were no signifimarital status, educaQOL-BREF Questionnaire cant differences between CLCP and non-CLCP groups in terms tion level, and sex of coping styles and quality of life. without CLCP and with a Polish ethnic background 100 healthy subjects GHQ-28, Taylor Manifest AnxiCLCP patients had significantly more psychiatric comorbidities ety Scale, Eysenck Personality than the control group. Sociodemographic attributes associInventory, Minnesota Multiphaated with high psychiatric morbidity include male sex, widowsic Personality Inventory hood, age group 30–34 yr, and unemployment. 25 children with no Harter Self-Perception Profile Children in the cleft group more often displayed “tongue out,” known physical “eye contact,” “mimicry, and “initiates conversation” facial abnormality behaviors. For the cleft group, “gaze avoidance” was significantly negatively correlated with social acceptance scores. The groups were comparable in their ability to pose and spontaneously express facial emotion.
75 randomly selected patients from a hospital
The CLCP group rated their life meaning, family life, and private economy significantly lower than the group without a cleft, but within normal limits. There were no significant differences between the two groups on the well-being scales. The overall level of quality of life was rather high in both groups. Pictorial Scale of Perceived There was no effect of cleft status on peer relationships. In the Competence and Social Acceptdirect observation components of the study, however, there ance for Young Children were difficulties in communication and socioemotional functioning in cleft patients compared with control patients. SF-36 Quality of life did not differ between bilateral CLCP group and the control group.
Quality of Life Survey
66 children, age- and sex-matched
Marcusson et al.48
Kapp-Simon et al.37
The effect of cleft lip on Murray socio-emotional funcet al.53 tioning in school-aged children Satisfaction with treatOosterkamp ment outcome in et al.49 bilateral cleft lip and palate patients Self-esteem, coping Pisula et al.50 styles, and quality of life in Polish adolescents and young adults with unilateral cleft lip and palate
Self-concept of primaryschool-age children with cleft lip, cleft palate, or both Quality of life in adults with repaired complete cleft lip and palate
113 patients recruited from members of staff
Specific Control Population Characteristics State-Trait Anxiety Inventory or No significant differences between those with CLCP and those State-Trait Anxiety Inventory without CLCP were noted in terms of state or trait anxiety. Parfor Children, Beck Depression ticipants with CLCP reported a greater number of depressive Inventory or the Children’s symptoms compared with participants without CLCP according Depression Inventory, Rosento the CDI, but no differences according to the BDI. Sixtyberg Self-Esteem Scale or the two percent of participants with CLCP reported having been Self-Esteem Index, Youth Selfteased, compared with 22% of participants without CLCP. Age Report or the problem scales of was a significant predictor for behavioral problems, happiness the Young Adult Self-Report with facial appearance, and satisfaction with speech. Overall, older subjects had more behavioral problems and were less happy with their appearance and their speech than were younger subjects. 172 normal children Primary Self-Concept Inventory Those with a cleft viewed themselves as less acceptable to peers, in elementary school more likely to require help, and more frequently sad/angry.
Self-reports of psychoHunt et al. social functioning among children and young adults with cleft lip and palate
Table 3. (Continued)
Volume 135, Number 1 • Cleft Lip and/or Palate Comparative Study
CLCP, cleft lip and/or palate; SDQ-1, Strengths and Difficulties-1; PEILCQ, Participation in Everyday Life Communication Questionnaire; SAD, social anxiety disorder; PHCSCS, Piers-Harris Children’s Self-Concept Scale; CDI, Child Depression Inventory; BDI, Beck Depression Inventory; SF-36, 36-Item Short-Form Health Survey; GHQ-28, General Health Questionnaire-28.
Child Behavior Checklist, Carter Children in both groups had normal psychosocial adjustment, Self-Perception Profile for but also reported moderate dissatisfaction with facial appearChildren/Adolescents, Social ance. Support Scale for Children and Adolescents, Children’s Health and Illness Profile, Social Skills Rating System Questionnaire, Satisfaction with Physical Appearance Scale 34 children with no known physical abnormality Slifer and Beck51 Self-concept and satisfaction with physical appearance in youth with and without oral clefts
Table 3. (Continued)
Specific Control Population Characteristics
Plastic and Reconstructive Surgery • January 2015 Behavior and Mood Disorders Psychosocial adjustment, behavior, and the presence of mood disorders were most commonly assessed using the Strengths and Difficulties Questionnaire (n = 7 studies).24–27,34,35,45 The Culture Free Self-Esteem Inventory (n = 3), SelfPerception Profile for Adolescents (n = 3), and 36-Item Short Form Health Survey (n = 2) were also used frequently. Factors that affect the presence of behavior and mood disorders include age and sex.30,44,54 Cleft-lip–cleft palate adult patients of either sex tend to have greater satisfaction with life compared with adolescents and do not differ in this metric from patients without clefts. Cleft lip and/or palate adolescent patients overall tend to exhibit significantly lower general and social selfesteem than control groups of the same age.30,44 In comparison with younger children, however, no definite trend exists as to which age group is more likely to experience psychological distress as a result of a cleft. With respect to sex, variable relationships exist, with some studies demonstrating improved self-concept and behavior among girls compared with boys, and others studies demonstrating opposite results.41,43 Ease of Survey Administration Of the 42 different instruments evaluated in this review, only the Pictorial Scale of Perceived Competence and Social Acceptance for Young Children was specifically determined to be inadequate in terms of detecting differences in perceived social competence and acceptance as discussed by Murray et al.53 Specifically, the authors state that this measure may not be able to accurately detect variations in the cleft lip and/ or palate population because of factors unique to the identity of this specific group. For other surveys, although there were difficulties noted when using the postal system resulting in low response rates and improper completion of the survey, no complications were noted with the actual surveys themselves in terms of content.25,29,49–51 The youngest reliable age for direct survey administration was approximately 5 to 6 years, and no maximum age limits were noted.
DISCUSSION In this systematic review, we identified 30 comparative studies of patient-reported outcomes among individuals with cleft lip and/or palate. The majority of studies used generic instruments, and few describe the specific psychometric properties unique to the instruments when applied to
Those with additional conditions reported significantly more psychosocial difficulties than children with a cleft alone. There were no differences in adjustment between children with a visible versus a nonvisible cleft. Children with a cleft alone reported significantly more positive social experiences than the children with a condition in addition to a cleft. In relation to a normal group, mean scores for children with a cleft alone were within the normal range for all subscales and were similar to mean scores from the reference group. The majority of children had medium or high self-concept. Global scores of selfesteem were above the mean for all groups, but there were differences based on sex and age. Adolescent girls experienced lower self-concept than younger girls. Adolescent boys experienced higher self-concept compared with younger boys. Anxiety and happiness were more problematic for girls than for boys regardless of age. Popularity was the only aspect that fell below the mean norms for all subjects. Sex and age together increased the predictive power for assessing the changes in self-concept. No significant differences between CLCP and cleft palate subjects. Older subjects rated themselves as less aggressive, less active, and having less somatic complaints than younger subjects. Male patients rated themselves as more aggressive than female patients in the CLCP group. No differences between male and female patients were noted in the cleft palate group.
Cleft lip/alveolus or CLCP, 132; cleft palate/syndromic cleft palate, 73
Cleft type, age, and sex differences in teen-agers’ ratings of their own behavior, self-esteem, and attitude toward clefting
CLCP, cleft lip and/or palate. Short-Form Health Survey.
Leonard et al.41
Self-concept of children and adolescents with cleft lip and/or palate
Missouri Children’s Behavior Checklist
Piers-Harris Self-Concept Scale
Strengths and Difficulties Questionnaire
In patients with clefts who were divided into resilient versus nonresilient personality types, there were no differences seen between the groups with respect to sex, visibility of cleft, or overall distribution of additional diagnoses. There was more teasing noted within the nonresilient group. Resilient children tended to be more satisfied with their speech and appearance compared with nonresilient children. Results indicated that visibility of the cleft did not have a substantial association with the child’s reporting of psychosocial resilience. There were no differences based on sex. In addition, having an additional diagnosis was significantly associated with higher degrees of cognitive dysfunction and general maladjustment. Girls aged 16 with a nonvisible cleft reported significantly less peer harassment and were more satisfied with appearance than the girls with a visible cleft. No group other than adolescent girls differed with respect to cleft visibility. Adolescent girls reported significantly more peer harassment than adolescent boys and were less satisfied with appearance. Children with a visible cleft were almost twice as likely to be teased compared with those with a nonvisible cleft. There was no association between satisfaction with appearance and levels of harassment in girls aged 10 yr. No differences were noted between girls and boys, or between children with a visible and a nonvisible cleft among the 10-yr-old age group. Choice of exclusion criteria affects the results obtained and thus may affect the conclusions drawn regarding this population. When scores for children with associated conditions were analyzed separately, those without additional conditions showed similar levels of psychosocial adjustment as national comparison samples.
Childhood Experience Questionnaire, Satisfaction With Appearance, Personality Inventory for Children
Toward a reconsideration of Feragan et al.34 inclusion and exclusion criteria in cleft lip and palate: Implications for psychological research When there is more than a Feragan et al.35 cleft: Psychological adjustment when a cleft is associated with an additional condition
There were no statistically significant differences between the cleft and control groups at any time points.
Social Avoidance and Distress Scale, Satisfaction With Life Scale, Culture Free Self-Esteem Inventory
Satisfaction With Appearance, Self-Perception Profile for Adolescents, Childhood Experience Questionnaire
Feragan et al. 43
Chua et al. 42
Peer harassment and satisFeragan and faction with appearance Borge32 in children with and without a facial difference
The comparison of psychological adjustment of patients with cleft lip and palate after maxillary distraction osteogenesis and conventional orthognathic surgery Social experience in 10-yearold children born with a cleft: Exploring psychosocial resilience
Table 4. Summary of Studies Using Subgroups of Cleft Lip and/or Palate Patients for Comparison with Other Subgroups of the Studied Population
Volume 135, Number 1 • Cleft Lip and/or Palate Comparative Study
Plastic and Reconstructive Surgery • January 2015 patients with clefts. Furthermore, the instruments used to measure patient-reported outcomes vary widely, and no accepted criteria standards exist. Although the overall number of studies on patient-reported outcomes among patients with cleft lip and/or palate is encouraging, the overall percentage of comparative studies in this body of literature is relatively low, suggesting a greater need for comparative effectiveness research to fully understand the implications of reconstruction in this patient population. Our results align with previous studies that have examined the use of outcomes assessment instruments in the cleft population and augment our understanding of how these measures can be systematically applied. In 2011, Klaasen et al. examined both patient- and proxy-reported outcome measures and classified outcomes into three main categories—physical, psychological, and social health constructs—providing a framework around which to develop new cleft-specific measures and evaluate existing nonspecific measures.21 Similarly, Eckstein et al. examined methods of assessing outcomes in cleft patients and identified a lack of comprehensive measures that capture both aesthetic and functional outcomes in this population.1 Importantly, we identify that the majority of studies do not use control groups for comparison, lack standardization of currently available tools, and are unable to be generalized across populations and age groups given the individualized form of survey administration used by each study. With regard to aesthetic, social, and behavioral outcomes, no consensus currently exists. Studies exist that demonstrate lower, equivalent, and higher self-reported perceptions among patients with clefts compared with those without clefts. Although it seems somewhat contradictory that patients with clefts are more satisfied with their appearance compared with patients without clefts, several reasons exist to explain this. First, children with clefts who are treated in a multidisciplinary clinic receive prophylactic and therapeutic intervention from psychologists, physicians, and social workers at an early age, whereas children without clefts do not commonly have access to such resources.25 These interventions could improve the coping mechanisms of adolescents and children to allow them to develop improved perceptions about their overall appearance. Second, surveys used to measure self-reported appearance may be less sensitive among children with clefts. For example, most surveys focus on appearance in general, and may not specifically address lip, nose, and midface deformities that are more prevalent among
children with clefts.21 Additional studies are needed to assess both specific and general aspects regarding satisfaction with appearance in this population. The lifelong impact a congenital deformity such as a cleft is an important consideration. The care for patients with a cleft progresses on a specific timeline from infancy to early childhood, with patients most commonly undergoing cleft lip repair at approximately 3 months of age, palate repair at 6 to 12 months of age, alveolar cleft repair at 6 to 9 years of age, management of velopharyngeal insufficiency beginning at approximately age 5, and skeletal and final nasal reconstruction in adolescence. A child’s need for reoperation or psychosocial intervention outside of these time points is ill defined. Given the risks associated with additional operations and the changes that occur with growth, revision surgery is taken very seriously and is performed only in specific circumstances, most commonly when patients themselves demonstrate aesthetic or functional dissatisfaction with their result. The question becomes how should patients with congenital abnormalities be evaluated longitudinally from birth through childhood and into adolescence. Although patient proxy measures must be used until the patient is able to read or understand pictorial representations of surveys, the accuracy of proxy measures must also be questioned, given that previous studies demonstrate differing perceptions between parents and children regarding the influence of the cleft on development. Furthermore, any assessment tool must be able to address the interplay between surgical outcomes, family dynamics, peer relationships, and psychosocial development. To improve our understanding of this patient population, we must work toward the creation of an appropriate evaluation strategy in a stepwise fashion to maximize accuracy and generalizability. By first focusing on self-reported patient outcomes, we can understand which factors of cleft care are most important for the patient, when revision surgery or psychosocial therapy type programs are necessary, and finally use patient-reported outcomes as the standard by which to design and measure patient proxy outcomes to more directly improve our understanding of younger age groups in the future. Our study has several notable limitations. As with any systematic review, our findings rely on the quality of the available literature. Significant differences existed between studies in terms of method of survey administration, patient population examined, and constructs assessed. Thus, it is difficult for us to quantitatively synthesize our data, given heterogeneity. Nonetheless, we were
Volume 135, Number 1 • Cleft Lip and/or Palate Comparative Study able to identify important themes in patient experiences following cleft repair, and applications and limitations of existing instruments. In addition, our findings are subject to publication bias, and we may have failed to capture comparative studies that remain unpublished. However, we performed a thorough search under the guidance of a research librarian of three comprehensive databases. Although we attempted to obtain full-text documentation for all examined abstracts, we did exclude studies that were available only in abstract form. Our review includes only those studies that use patient-reported outcomes and excludes those that use only proxy-reported outcomes. Although this inherently limits our ability to understand outcomes in the youngest patients with clefts, it also improves our ability to detect variations in development during times in the patients’ lives when enrollment in specific forms of psychosocial intervention and consideration for revision surgery are most important. In addition, by identifying the best method of patient-reported outcome assessment, we can also use these measures as patient proxy assessments in the future. Despite these limitations, our findings provide important insight regarding comparative evaluation of outcomes among individuals with cleft lip and/or palate. To date, current patient-reported outcome assessment tools have not been systematically applied, limiting the ability to compare findings across studies. Given the multitude of instruments currently available, it may be more efficient to identify those tools with adequate validity, reliability, and responsiveness over the creation of new instruments, which requires numerous resources and time. Accurate measurement of outcomes among children with cleft lip and/ or palate is necessary to critically appraise the effectiveness of current treatment options, longitudinally assess patient adjustment over time, and identify benchmarks by which the quality of care of these patients can be improved. An example of a single, generic instrument that can provide a comprehensive and sensitive assessment of outcomes is the Patient-Reported Objective Measurement Information System that uses item response theory to assess outcomes across various conditions and scenarios. In 2004, the National Institutes of Health initiated a collaborative effort to develop a national system to measure health status using generic items that can be applied across a range of conditions using comparative population norms. Using advances in psychometric theory, the Patient-Reported Outcomes Measurement Information System can
be adapted to a computerized framework to measure health status with less responder burden and greater discriminative ability compared with previous, or legacy, instruments. Among children, the Patient-Reported Objective Measurement Information System item banks have been developed for fatigue, anxiety, depression, and peer relationships, and have been adapted for either self or proxy report. The Patient-Reported Outcomes Measurement Information System can facilitate survey administration, given the ease of use and practical, technologically innovative format; it has already been successfully used to assess long-term health status and treatment effectiveness in multiple pediatric conditions, including upper extremity injuries, asthma, and psychosocial illness.55–58 Instruments such as the Patient-Reported Outcomes Measurement Information System and other such analogues may represent an opportunity for better patient-reported outcome assessment in the future.
CONCLUSIONS Many surgical techniques exist to reconstruct cleft lip and palate. Although surgery can provide dramatic restoration of form and function, many children live with asymmetry and deformity and often experience lifelong difficulty with social relationships, self-esteem, and psychological functioning.59 In this systematic review, we identified inconsistency regarding instrument use and a lack of consensus regarding outcomes measurement in this patient population. Going forward, optimizing comparative studies of individuals with cleft lip and/or palate can allow us to target surgical management, streamline multidisciplinary team interventions, and implement changes in policy according to individualized patient data and development. Jennifer F. Waljee, M.D., M.S. Section of Plastic Surgery University of Michigan Health System 2130 Taubman Center, SPC 5340 1500 East Medical Center Drive Ann Arbor, Mich. 48109-5340 [email protected]
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